Head and neck cancer treatment outcome priorities: A multi-perspective concept mapping study

With the increasing focus on patient-centred care, this study sought to understand priorities considered by patients and healthcare providers from their experience with head and neck cancer treatment, and to compare how patients’ priorities compare to healthcare providers’ priorities. Group concept mapping was used to actively identify priorities from participants (patients and healthcare providers) in two phases. In phase one, participants brainstormed statements reflecting considerations related to their experience with head and neck cancer treatment. In phase two, statements were sorted based on their similarity in theme and rated in terms of their priority. Multidimensional scaling and cluster analysis were performed to produce multidimensional maps to visualize the findings. Two-hundred fifty statements were generated by participants in the brainstorming phase, finalized to 94 statements that were included in phase two. From the sorting activity, a two-dimensional map with stress value of 0.2213 was generated, and eight clusters were created to encompass all statements. Timely care, education, and person-centred care were the highest rated priorities for patients and healthcare providers. Overall, there was a strong correlation between patient and healthcare providers’ ratings (r = 0.80). Our findings support the complexity of the treatment planning process in head and neck cancer, evident by the complex maps and highly interconnected statements related to the experience of treatment. Implications for improving the quality of care delivered and care experience of head and cancer are discussed.


Introduction
Head and neck tumours affect sensitive anatomical structures responsible for vital functions, such as speech, chewing and swallowing.Although treatments have shown promising oncological outcomes, they often have an impact on a person's physical, psychological and social aspects of life [1,2].Thus, healthcare providers (HCP) have been navigating the balance between cure and preserving quality of life [3], which in turn makes treatment planning for head and neck cancer (HNC) a complicated process.The process gets more challenging as, currently, patients are expected to be part of treatment planning and decision making.In this regard, it is valuable to understand patients' perspectives around their experience with treatment, and how they might compare to the perspectives of HCPs.
Head and neck cancers are treated either by surgery, systematic therapy such as chemotherapy, radiation therapy, or some combination thereof [4].In most cases, treatment can have distinct short and long-term toxicities affecting different aspects of an individual's life.The use of multiple treatment modalities can further aggravate these toxicities [4][5][6][7][8].Known structural and functional outcomes include disfigurement, scar formation, skin irritation, tissue fibrosis, lymphedema, hair loss, pain, and hyperalgesia.Oral intake and maintaining nutrition also can be affected by xerostomia, oral mucositis, trismus, dysphagia, dysgeusia, nausea, malnutrition, and weight loss.Communication might be impacted by either voice loss, speech disorder, hearing loss, tinnitus, vertigo, osteoradionecrosis, and fatigue [1,6,9,10].With all these changes in physical functions, a deterioration in psychological wellbeing of individuals is anticipated [11].People with history of HNC present with the highest levels of distress when compared to other cancers [12].It is also known that anxiety, depression, fear of relapse, low self-esteem and embarrassment are expected [11,13].Consequently, a decrease in social interactions is seen [1,11,13], highlighting the global impact of this type of cancer and its treatment on individual's life.
Considering the impacts of treatment on a patient's life, HCPs have been increasingly acknowledging quality of life assurance during treatment planning, along with ensuring cure and survival [3,14].With the increasing focus on person-centred care, understanding patients' perspectives became more important and including their perspective became necessary.In this regard, previous studies explored patients' priorities and found that survival from cancer was their top priority [3,15,16], and that patients were inclined to accept treatment toxicities for survival [17].Physical functions however, were still of high priority to patients.Swallowing for example, was ranked as the third highest priority following cure and survival in one study [18].Thus, there appear to be competing priorities that still need to be more fully understood in the context of the experience of HNC.Recognizing the outcomes and priorities of those affected can be valuable to improving the quality of care and can inform future treatment planning.
To our knowledge, existing patient priorities are focused on functional outcomes, and little is known around perspectives on psychosocial and/or other aspects of outcomes [19].The specific focus of priority studies on functional outcomes could be attributed to the fact that most priority measures were developed by investigators and presumed to include patients' perspectives around the treatment of HNC.Yet, experiencing HNC diagnosis and treatment might include other valuable considerations that investigators might be missing when developing priority measures without initial input from patients.To help identify these considerations, it is imperative for patients to identify their priorities from their own experience.

Purpose
The primary purpose of this study was to explore patients' perspectives on HNC treatment and to actively identify their considerations by eliciting an exhaustive list of outcomes and considerations associated with their treatment experiences.We also sought to understand how these considerations are valued in terms of priority, and how they compare to HCPs' priorities.Developing a comprehensive understanding of patients' experience in this way will contribute to future treatment planning and enhance care experience for future patients.

Methods
This exploratory, mixed-method study was ethically approved by the Health Research Ethics Board of Alberta: Cancer Committee (HREBA-CC-19-0012).All data collection was completed using CS Global Max™, a secure web-based platform for group concept mapping.Consent was indicated by overt action when participants submitted their responses online.Participants were able to withdraw responses up to the point of clicking the button "save and submit".All responses were anonymous and therefore irretrievable once submitted.

Participants
Convenience and snowball sampling methods were used to identify potentially eligible participants from our targeted population.Patients eligible for participation were adults 18 years or older, who were diagnosed with squamous cell HNC, were either receiving treatment or were post-treatment at the time of study enrollment, with treatments being delivered in Alberta, Canada.Patients with thyroid or salivary gland cancer (adenocarcinoma), and/or who did not start treatment, and/or patients with conditions that could compromise their ability to perform the required task in the study (e.g., dementia), were excluded from the study.Healthcare providers were individuals who regularly work with HNC patients in Alberta.

Recruitment
Nine-hundred-and-eighty-five patients met the eligibility criteria within the Alberta Cancer Registry (ACR) database.The ACR directly mailed out study information packages that included the study information letter, ACR cover letter, and ACR information pamphlet to all potential participants.The study information letter included a direct link and a Quick Response (QR) code to scan and access the online platform and participate in the study.Patients and HCPs also were recruited through snowball recruitment methods including word of mouth.Posters (including the QR code) were displayed in public spaces such as hospitals and clinics where the target population receives consultation and treatment in Edmonton and Calgary.Recruitment took place between September and November 2019.

Data collection
We used an exploratory, descriptive design using a mixed-method approach called Group Concept Mapping (GCM).The use of GCM was facilitated using the Concept Systems Global Max™ software (www.conceptsystems.com)as a web-based platform to collect and analyze data.A preparation stage prior to active data collection was undertaken to develop and test the prompt utilized in the data collection phase.Data collection in GCM is comprised of three activities: brainstorming, sorting, and rating [20].In this study, data were collected in two phases; phase one (brainstorming activity) and phase two (sorting and rating activities) (Fig 1).These phases were conducted at separate time periods to reduce participant fatigue and attrition.In each phase, participants completed a questionnaire capturing demographic and clinical characteristics.

Preparation for phase one
The research team identified the purpose of the study, target population, and developed and pilot tested the prompt used in the study.Developing the prompt is an essential step in GCM as it affects the outcome of the study [21].The more accurate the prompt, the more accurate the elicited responses will be, and this can be determined by pilot testing the prompt on a group of representative target participants to ensure that it results in relevant statements [20].The initial prompt was pilot tested on patients and HCPs with experience of HNC.The final crafted prompt was: "Important considerations throughout the entire experience of head and neck cancer are.."

Phase one
Phase one was comprised of an activity called brainstorming.Demographic questions were asked to capture the demographic and clinical characteristics of participants.In this activity, participants were instructed to provide as many statements as they could in response to the focused prompt (stated above).Brainstorming was estimated to take about 20-40 minutes to complete.This phase was open between September 16, 2019 and October 20, 2019.
Once brainstorming was closed, the statements collected underwent a structured and iterative synthesis process by research team, as described by Kane and Trochim [20].All generated statements were reviewed following a structured synthesis process to remove redundancies from the brainstorming activity, in order to create a manageable number of concise and easily comprehended statements that would be carried forward to the sorting and rating activities.Previous literature supports the use of 100 statements or less to sufficiently cover the variety of thoughts while being manageable for participants in the sorting and rating activities.[20,22] In brainstorming, the quality of data depends on the data saturation rather than number of participants [20].As stressed in the literature [20], we kept the edits at minimum, maintained the original meaning of the statement, and ensured that no ideas or thoughts have been removed during this process.

Phase two
Demographic questions were asked to capture the demographic and clinical characteristics of participants.This phase included a sorting activity where each participant grouped statements together in terms of what they perceived to be similarity in theme.They then provided a label for each group that described a shared theme.This activity enabled us to better understand how participants perceive the interrelationship between statements.Participants received a "thank you" message upon submitting their final response in the sorting activity and a reminder to proceed and respond to the rating activity, with a reminder of the last day to submit the response for phase two.Phase two also included the priority rating activity where participants rated each statement separately using a Likert-scale (1 to 5), with 1 being not important at all, and 5 is extremely important.This phase was estimated to take between 60 and 90 minutes in total.Participants could pause at any time and return to complete their response to the activity.The phase was open for three weeks, from November 9-30, 2019.Participants were provided with a link and login information.They were able to log in anytime within those three weeks.

Data analysis
Data were analyzed using the (CS) Global Max™ software.The independent variable was participant group.Dependent variables included: statements, statement sorting results, and the rating of each statement.Covariates included: age, sex, marital status, education level, tumor site, stage of cancer at diagnosis, time since treatment, human papillomavirus (HPV) status, and treatment modality.
All statements from the brainstorming activity were converted to quantitative data by core mapping analysis.In this analysis, each statement was numbered and entered into a cross-correlational matrix.From there, a tally of the number of times two statements occurred together was created.A series of analyses took place within the software after the creation of this matrix [20].The reader is referred to (S6 Appendix) for a detailed description of these analyses.
Nonmetric multidimensional scaling of the matrix was performed to site each statement as a (x,y) point on the point map and depict the relative distance between statements.Stress value was used to assess the goodness-of-fit of the resulting map, with lower values indicating better fit, with an average of 0.285 and range (0.205-0.365) being suggested in the literature as target values [20].The (x,y) points on the point map underwent a hierarchical cluster analysis that identified the representation of how statements were grouped together in terms of similarity.Each possible cluster map with clusters 6-10 was examined using the cluster and statement bridging values, along with interpretation of cluster contents to identify the final number of clusters that best fit the results.The multidimensional maps generated by the software were the visual representation of the project's data and finding.They depicted how the participants perceived the statements in terms of similarity, and their importance,

Phase one
A total of 63 participants (n = 59 patients, n = 4 HCPs) completed phase one.Detailed participant demographic and clinical characteristics are reported in Table 1.Two hundred fifty statements were generated.Data variety was ensured by the number of statements generated and a wide range of perspectives captured.For a successful synthesized set of statements, the number of statements was reduced to 94 statements after the synthesis process without comprising any ideas generated.This number falls under the average reported number of statements found in the literature of 96 statements (Rosas & Kane, 2012).Most of the statements were semantically and syntactically preserved from the original response gathered.

Phase two
A total of twenty-four people (could be different than those participated in phase one) participated in phase two (n = 15 patients, n = 9 HCPs) and completed either sorting and/or rating activities.Details of participants' demographics in Table 1.Based on the literature, 20-30 sorters in total reflect the diversity of perspectives and is sufficient to reduce the stress value and provide reliable maps [23].Sorting.In phase two, 22 out of the 24 participants completed the response sorting task.One participant's sorting responses were removed because the sorting was based on what the outcome meant to the individual (e.g., not an issue for me, not a serious issue for me, major concerns) rather than categorizing statements into groups based on their conceptual similarities.Thus, 21 sorters' responses were identified and eligible for core analysis.
The multidimensional scaling (MDS) analysis of sorting data generated the point map from which all further analyses were based (Fig 2).All 94 statements are represented on this map, as each point represents a statement/outcome.A final MDS stress value of 0.2213 was obtained.Following that, a cluster analysis of the 94 (x,y) point map was examined via cluster bridging values along with the cluster replay map and the research team's judgement to identify the final number of clusters and for or our purpose, we considered an eight-cluster structure as a best fit to our results (Fig 3).Table 2 provides description of each cluster.The central location of cluster 4 'Nutrition' reflects the relationship of the statements in this cluster with different aspects and statements from other clusters.Survival might be located at this area of the map, not because of how much it is connected to nearby statements of 'Nutrition' but rather because it might be related to several other areas and clusters on the map.
Rating.Twenty-three out of the 24 participants completed rating for all 94 statements.The average rating was computed for each statement and for each cluster of statements.For both groups combined, the top five ranked singular statements in terms of importance (i.e., average ranked scores out of 5) identified at a statement level were 'promptness of treatment' (4.91), 'promptness of diagnosis' (4.86), 'knowledgeable and experienced healthcare providers in head and neck cancer' (4.82), 'survival' (4.68), and 'clear detailed upfront information of the case and treatment plan' (4.64).
The highest rated clusters for both groups combined in terms of importance were 'Education', with an average rating of (4.28), 'Person-Centred Care' with an average rating of (4.20), 'Nutrition' with an average rating of (4.15), and 'Continuing Care' with an average rating of (4.08).The average rating values of the clusters are plotted in the cluster rating map (Fig 4).
The pattern match (Fig 5) represents the level of agreement on the importance of outcomes (clusters) between patients and HCPs.Overall, there was a strong correlation between patient and HCPs' ratings (r = 0.80).However, although 'Psychosocial Concerns' was the third highest-ranked priority for HCPs, it was the fifth most important consideration for patients.This difference was found to be statistically significant (t (14) = 2.76, p <0.02).

Discussion
In this study, we aimed to explore the HNC treatment experience and priorities from the perspective of those affected.Our first objective was that participants identify treatment outcomes and considerations in a brainstorming activity that would then be rated in terms of priority.As a result, our findings from the brainstorming activity revealed treatment experience-related considerations that were reported previously in the literature, and others that were not investigated previously in terms of their priority.We also measured priorities for patients and HCPs  at a statement level, revealing the following top priorities: timely diagnosis and treatment, having HCPs experienced with HNC, survival, and having clear upfront information on treatment.We then measured priorities at a cluster level to understand how general themes might differ in priority.The 'Education' cluster was the highest rated cluster.Furthermore, we explored the relationship between patients' and HCPs'priorities to help us understand how to enhance future treatment planning and care experience.Findings revealed an alignment of most priorities between the two groups, except for a significant difference of priority rating in the 'Psychosocial Concerns' context.The findings of this study bring new information to the literature that may suggest that previous consideration of priorities has been narrow in focus.Specifically, in the present study, when priorities were identified patients' perspectives, they were holistic of the care experience and not specific to treatment-related outcomes or side effects.Thus, while treatment side-effects are a reality of the condition of HNC, they should be considered in a wider context of the health care experience.

Patient-reported outcomes in the health care context
In examining treatment outcomes and priorities, previous studies used measures developed by investigators [24,25].It is now well-recognized in clinical practice and research that patients' perspectives are essential from the outset to fully understand their care experience and assess treatment options [24,25].In our study, we wanted to explore if there are valuable priorities that are missed by investigators and which can only be identified by individuals who experienced HNC treatment.To that end, patients and HCPs in our study actively identified a holistic set of statements/ considerations related to their treatment experience, and were refined from a brainstorming activity.These included statements not only related to treatment outcomes but also to healthcare-related, reflecting the global and complicated impact of this illness.Treatment outcomes reported in the literature that also were identified in the present study included: survival [3,15,26]; pain and functional outcomes [3,15,26,27]; physical alterations and psychological outcomes [1,2]; social impact of treatment [1,11,13]; financial challenges [28,29]; support and involvement in decision-making [30].Taking higher priority, however, were healthcare-related considerations centered around the care experience including accessibility to health services for those in rural communities; care services after primary intervention; psychosocial support before, during, and after treatment; HCPs' knowledge; and patient and family education.The resulting maps provided visuals on how outcomes related to healthcare experience and services were more important for both groups than treatmentrelated outcomes and side-effects.This might reflect how meaningful it is for people with history of HNC to have a positive care experience, and might reflect the importance that education and support have on their care experience.

Priority ranking of healthcare-related considerations
As we explored priority ranking among both groups, we found that promptness of treatment, education, and support were the most valued considerations.

Timeliness of care
At a statement level, promptness of treatment was ranked the highest for both patients and HCPs.Despite recognizing the need for prompt HNC intervention in ensuring optimal outcomes, delays in delivering care for HNC are frequently reported [31,32].These delays ultimately affect functional outcomes [32,33] and mortality [32,34].Barriers to timely treatments still exist [35], and successful strategies to facilitate the promptness of HNC treatment remains undetermined [32].Within the context of planning and providing timely treatment, delivering information around treatment plan and outcomes also was highly valuable for our participants.

Education
While recognizing promptness of treatment is a priority, participants also identified an important consideration to their treatment experience, which was education.Education, as a theme or cluster, was the highest ranked among our participants.The results of our study further showed that there were two categories of information that patients and HCPs identified as critical to the experience of HNC treatment.The first was providing education on the treatment options and what the treatment will entail, and the second was on the potential long-term side effects that patients may encounter and how to manage and cope with these effects.The value of these types of information has been substantiated in the literature in several studies as it highlighted the impact on overall outcomes [36].Besides acknowledging the value of such information, considerations around a patient's emotional readiness to comprehend information is also critical.
Patients have different preferences regarding when to receive information around their treatment.In previous research, patients have stressed the importance of receiving thorough information on all expected outcomes before initiating treatment to better anticipate and prepare for possible side effects [36,37].While some patients preferred to know everything at the time of diagnosis, others felt it was difficult to comprehend information on side effects when provided right after diagnosis [37], and others reported difficulty remembering information provided before treatment sessions regarding their treatment plan [37].Consequently, patients reported the value of having an accessible resource for information.In the age of the internet, patients seek health information related to symptoms, etiologies and treatment options online and even try to educate themselves in interpreting test results [38].Such health behaviors pose a risk that patients may rely on nonmedical sources to inform their decision-making.This highlights the need for trustworthy and informed sources to provide appropriate information to patients and their families, in a timely manner [30,36,39] and aligns with the priority found in our study 'providing an accessible resource/tool for information and common questions'.Our findings point to the need for establishing strategies to support communication, deliver information continuously, and to outline pathways for following-up with patients throughout their treatment experience.

Person-centred care
Developing strategies for communication should consider that successful patient-clinician interaction is a part of the support that patients need to be involved in their care.This in turn is the cornerstone of a person-centered approach of care.The 'Person-Centred Care' cluster in the present study included statements around involving the patient and their families in care decisions, support, and knowledge.
Participants in our study reported that 'being involved in decision-making' and 'keeping the family informed' are important when planning treatment for HNC.Patients generally consider it highly important to include their families and friends in the decision-making process and consider their opinions and concerns throughout the treatment process [30].Yet, not having sufficient information on the treatment options makes it difficult to be actively involved in decision-making [40].Thus, to be able to discuss and share thoughts around decisions related to care, patients need to be educated on the nature of their illness, their care options, prognosis and expected outcomes, and to be supported to share thoughts and concerns during their care process.
As a form of support, patients appreciate HCPs showing empathy and care, as well as those who communicate actively with them [36].'Communication with healthcare providers' and 'Engaged healthcare providers with compassionate care' were high priorities in the 'person-centred care' cluster in this study.Patients need to communicate effectively with HCPs to be able to make decisions.They also need to be comfortable to provide feedback on treatment options, outcomes and share their concerns following treatment, which may include physical, functional and psychosocial impacts.

Psychosocial concerns
The psychosocial impacts following cancer treatment were identified by our participants as an area to be considered when planning treatment.Although it was the third priority for HCPs, it was only the fifth important consideration for patients.Different interpretations might justify this significant difference of rating.Healthcare providers have access to research focused on psychosocial impact of treatment.One study, for instance, reported that two out of the four highest suicidal populations with cancer are those with history of oral cavity and laryngeal cancers [41].The continuous exposure to similar reports might have an influence on HCPs' perspective.Moreover, the topic of clinical empathy has been getting attention in recent years, and training to improve skills of empathy in HCPs has been supported [42].This focus is derived from the positive relationship found between clinical empathy and improved care.It also was reported that with empathy, patient-clinician interaction is enhanced and patients therefore become more open to share their worries and concerns [42].The focus on empathy and the patient-clincian interaction could explain why HCPs perceive psychosocial concerns as a high priority [40].
Psychosocial impacts of cancer treatment are highly prevalent in individuals with HNC and these outcomes were important for patients in our study.Yet, it was interesting to see that continuing care after treatment was of a higher priority to them, reducing the ranking of psychosocial concerns.This might reflect how patients focused their priority rating on accessibility to care, care and support services, education and resources, more than anatomical, functional, and psychosocial treatment impacts.This significant difference in priority requires further investigation in future work.

Limitations
There are several limitations to the study that may limit the generalizability of the findings.Although the sample was sufficient to produce valid and reliable data in GCM, it was small to be generalizable for all patients and HCPs who have experience with HNC.The small sample of participants with different types of HNC limits the ability to perform sub-analysis based on each type.Participants in this study experienced treatment only from the province of Alberta and because healthcare is provincial in Canada, our findings from Albertans may not be representative of patients in other geographical regions.A larger study can consider the priorities of HNC patients globally.This would allow for further investigation of underlying factors causing the differences in priorities.Other limitation related to study design is noted in the wording of the rating question, the question stated to rate statements in terms of "importance", which can be perceived differently than "priority" and thus perhaps have caused participants to rate statements differently.
Although one of the objectives of the study was to compare the priorities of patients and HCPs, there might be a selection-bias where a higher number of patients participated in the rating activity compared to HCPs.This although did not affect the significance of the analysis and findings, it may present some bias toward the conclusion.Non-respondent bias could be present in this study, given that patients with psychosocial may not be as motivated to participate in a study.Thus, cautions should be taken with generalization of findings and conclusions might take this into consideration.
Despite the fact that this GCM facilitated the active identification of treatment considerations from participants and allowed the capture of multiple perspectives, it does not provide us with a deep understanding of why some outcomes are of a higher priority than others.Future studies might consider conducting qualitative interviews or focus groups to further understand the participants' priorities.Of particular importance would be to understand why education and person-centred care are higher priorities than side-effects.Are these priorities unmet?Or otherwise met and believed to be factors for a successful experience?Further research may investigate details of priorities in each phase: diagnosis, treatment, and transitioning to rehabilitation.
Another study limitation was the low response in the online method used.Although online methods of GCM have certain benefits including access to many participants, a low response rate has been addressed in the literature and noted in our study [20].Lastly, one of the outcomes extracted from brainstorming activity (i.e., "tongue pain") was accidently duplicated in the synthesized set, and participants had to sort and rate this statement twice.When the software experts were consulted, the duplication issue was found to be somehow frequent, and since the duplicated statement was found together in the same cluster after analysis, no negative effect is estimated.

Conclusion
Person-centred care requires an understanding of patient needs and priorities.Based on the results of our study on priorities, multiple considerations should be taken into account to improve the HNC care experience.The perspective of patients and HCPs' priorities were largely in agreement.Timely care, education, and person-centred care were identified as the most meaningful considerations for future treatment planning.

Fig 3 .
Fig 3. Cluster map.This figure depicts the eight clusters of statements.Smaller clusters are more focused in concept while larger clusters are relatively heterogeneous.https://doi.org/10.1371/journal.pone.0294712.g003

Fig 4 .
Fig 4. Cluster rating map.This figure illustrates the clusters in layers reflecting average rating.The more layers, the higher the average rating is given to the cluster in terms of priority.https://doi.org/10.1371/journal.pone.0294712.g004

Table 1 .
(Continued) Denote multiple options apply.Participants were able to choose more than one tumor site and/or treatment type as it applies to their case.

Table 2 . Cluster descriptions.
7Statements in this cluster reflected physical and functional impacts of treatment that are of a persistent nature or with uncertain interventions available for them.https://doi.org/10.1371/journal.pone.0294712.t002